Xu Gang, Amei Amei, Wu Weimiao, Liu Yunqing, Shen Linchuan, Oh Edwin C, Wang Zuoheng
Department of Mathematical Sciences, University of Nevada.
Department of Biostatistics, Yale School of Public Health.
Ann Appl Stat. 2024 Mar;18(1):487-505. doi: 10.1214/23-aoas1798. Epub 2024 Jan 31.
Many genetic studies contain rich information on longitudinal phenotypes that require powerful analytical tools for optimal analysis. Genetic analysis of longitudinal data that incorporates temporal variation is important for understanding the genetic architecture and biological variation of complex diseases. Most of the existing methods assume that the contribution of genetic variants is constant over time and fail to capture the dynamic pattern of disease progression. However, the relative influence of genetic variants on complex traits fluctuates over time. In this study, we propose a retrospective varying coefficient mixed model association test, RVMMAT, to detect time-varying genetic effect on longitudinal binary traits. We model dynamic genetic effect using smoothing splines, estimate model parameters by maximizing a double penalized quasi-likelihood function, design a joint test using a Cauchy combination method, and evaluate statistical significance via a retrospective approach to achieve robustness to model misspecification. Through simulations we illustrated that the retrospective varying-coefficient test was robust to model misspecification under different ascertainment schemes and gained power over the association methods assuming constant genetic effect. We applied RVMMAT to a genome-wide association analysis of longitudinal measure of hypertension in the Multi-Ethnic Study of Atherosclerosis. Pathway analysis identified two important pathways related to G-protein signaling and DNA damage. Our results demonstrated that RVMMAT could detect biologically relevant loci and pathways in a genome scan and provided insight into the genetic architecture of hypertension.
许多基因研究包含有关纵向表型的丰富信息,这需要强大的分析工具进行最佳分析。纳入时间变化的纵向数据的基因分析对于理解复杂疾病的遗传结构和生物学变异至关重要。现有的大多数方法都假定基因变异的作用随时间恒定,无法捕捉疾病进展的动态模式。然而,基因变异对复杂性状的相对影响会随时间波动。在本研究中,我们提出了一种回顾性变系数混合模型关联检验(RVMMAT),以检测对纵向二元性状的时变基因效应。我们使用平滑样条对动态基因效应进行建模,通过最大化双重惩罚拟似然函数估计模型参数,使用柯西组合方法设计联合检验,并通过回顾性方法评估统计显著性,以实现对模型错误设定的稳健性。通过模拟,我们表明回顾性变系数检验在不同的确定方案下对模型错误设定具有稳健性,并且比假设基因效应恒定的关联方法更具功效。我们将RVMMAT应用于动脉粥样硬化多族裔研究中高血压纵向测量的全基因组关联分析。通路分析确定了与G蛋白信号传导和DNA损伤相关的两个重要通路。我们的结果表明,RVMMAT可以在全基因组扫描中检测到生物学相关的基因座和通路,并为高血压的遗传结构提供见解。